Veera Jayachandra Allu
Consultant, Department of General and Emergency Surgery
William Harvey Hospital, East Kent Hospitals University NHS Foundation
E-mail: [email protected]
Received: July 30, 2020; Accepted: August 20, 2020; Published: August 31, 2020
Citation: Allu VJ, Basu S, Chowdhury A, Shrestha A, Shah A (2020) Acute Appendicitis in De Garengeot Hernia – A Systematic Review. J Univer Surg. Vol.8 No.4:3
Introduction: Presence of appendix within a Femoral Hernia is a rare pathology named eponymously as De Garengeot’s hernia that is mostly identified as an incidental finding during exploration for an incarcerated Femoral Hernia and even rarer is the incidence of acute appendicitis in these cases.
Aim: To undertake a systematic review of the published case reports, focussing on the incidence of acute appendicitis within a De Garengeot hernia, the relationship between pre-operative diagnosis and surgical technique, as well as the incidence and surgical outcome.
Materials and Methods: A PRISMA modelled literature search was carried out across PubMed, ProQuest, and BMW Case Report databases with the search terms ‘Femoral Hernia’, ‘Appendix’, ‘De Garengeot’s’ in various combinations, limited to English language, published between 1960 to 2019. Authors report another case report, not included in the systematic review.
Results: Systematic review identified 83 published data reporting 111 cases. 74 (66%) cases presented with painful groin swellings. Computerised Tomography performed in 43 (38%) accurately diagnosed De Garengeot’s in 32 (74%) patients. The most common surgical approach was Lockwood’s low approach 35 (31%) followed by the Lotheissen’s approach 23 (21%). 81 (72%) underwent herniorrhaphy with non-absorbable sutures and 20 had mesh repairs (18%). Ten (9%) patients were reported to have postoperative morbidity with wound infection being the most common complication and one recorded death.
Conclusion: Rarity of De Garengeot’s hernia is shown by the limited availability of data that restricted the author’s ability to conclude a single diagnostic pathway or a specific surgical technique for this condition. CE CT is shown to be a relatively accurate form of imaging tool, as well as Ultrasound scans in the event that there was accessibility issue. Sequestrated appendicectomy and hernia repair via laparoscopy could be an option; however, there is lack of data to warrant its effectiveness as the surgical option of choice over an open groin approach in the presence of an incarcerated groin swelling.
Surgery; Colorectal; General Surgery
Caecal appendix appearing inside a Femoral Hernial sac was first described by the French surgeon René Jacques Croissant De Garengeot  in 1731 and is eponymously named after him (in 2005 by Akopian G et al. ) and even rarer is the incidence of an appendicitis reported as low as 0.8% - 0.13% . Femoral Hernia occurs as a result of the protrusion of the peritoneal sac with/without abdominal contents through the femoral ring into the femoral canal, medial to the femoral vein and below the inguinal ring . It is described as the third most common type of hernia, with a higher incidence in women (20%) than men (5%) . The rigid and narrow femoral neck increases risk of strangulation (15% -20%) In rare cases, the appendix can travel and be a content of the Femoral Hernia, reported in 0.5% - 5% of strangulated hernias.
De Garengeot Hernia remains an incidental finding and is often approached as a strangulated hernia prior to intervention. There is no prescribed diagnostic methodology either. A systematic review for this rare surgical condition was undertaken to identify the reported incidence of acute appendicitis in a De Garengeot hernia, tools used for pre-operative diagnosis, the surgical technique applied and their outcome.
A literature search with terms ‘De Garengeot’, ‘Femoral Hernia’, and ‘Appendicitis’ was undertaken on PubMed database for any publication in the English Language, between 1960 and 2019. The search results returned studies published after 2005, given that the term ‘De Garengeot’ was associated with the condition in 2005 . Amending the search with only ‘Femoral Hernia’ and ‘Appendicitis’ identified earlier published case reports, with a total of 135 articles available with full text. Another search was run in ProQuest, with the terms ‘Femoral Hernia’, ‘De Garengeot’, ‘Femoral Appendicitis’, ‘Appendix’ and ‘Appendicitis’ with the search limited to English language peer-reviewed case reports published after 1970 that resulted in 68 published articles. A further manual search through the references of the published articles was also undertaken to find any further case reports. The results were screened for duplications and other factors as outlined in the PRISMA flow diagram (Figure 1).
Figure 1: PRISMA flowchart
A total of 83 studies that fulfilled the criteria were analysed for patient demographics, presentations, diagnostic evaluation tools, surgical approach, and post-operative outcome. Surgical approach was categorised in relation to the inguinal incision: Lockwood's infra-inguinal, Lotheissen's trans-inguinal and Mc Evedy's high approach , unless specific techniques were mentioned. Any incision away from the inguinal was categorised as a laparotomy.
Publication bias - Data taken from published case reports risks significant publication bias as there will be reluctance for publication for cases with unfavourable outcomes. To counter the publication bias, two of the authors ran independent searches and compared results. Manual searches were also done in ‘Grey Literature’ (other searches in PRISMA diagram), such as working papers and scholar publications in addition to the popular journals to increase available data. Only peer reviewed articles were included in the final search, excluding any articles that the authors could not get full access to.
Total 111 patients were reported across 83 published articles [7- 87]. The author’s case report is not included in the systematic review. Of the 83, 70 published articles were single case reports, while the remaining 13 involved multiple cases.
Voitk AJ et al.  reported one chronic inguinal sinus of over two years in the two case studies reported. The chronic case was excluded to avoid skewing of the data, with the other case in the study retained.
The results are compared by totals and means when possible. Data is collated in tabular forms to summarise the interventions.
The most common presentation among these case reports was sudden onset painful groin swelling in 74 (66%) patients within which 88% (n=65) had no associated nausea or vomiting; 15 (13%) patients presented with painless groin swelling as their primary presentation and only 2 (0.2%) presented with diarrhoea. Mean duration of the symptoms was 4 (0-7) days for patients with acute presentation. Eight patients reported symptoms of pain longer than 10 days, with one patient reporting pain for 42 days. Out of 111 patients, 103 patients were identified by gender and of these 103 cases, 81% were females.
Clinically, 79% of the patients (n=91) had presented with a groin swelling including 49 with associated erythematous skin changes. 46% (n=51) of the patients were febrile on admission and 4 had features of shock. Eight had undergone previous hernia repairs (Table 1).
SymptomsNumber of Cases*%Swelling in Right Groin9182%Groin Pain at Presentation8173%Fever5146%Nausea at Presentation2623%Abdominal Pain1413%No Pain1614%
*Some patients exhibited multiple symptoms. Each presenting symptom is counted per patient at initial observation.
Table 1: Initial presenting symptoms in reviewed publications.
Blood investigations were performed in 92 (82%) patients. In 47 (51%), there was reported leucocytosis and elevated CRP and the remaining 45 (49%) showed normal blood picture. 71% (n=76) of the reported case studies had additional investigations, with some patients having undergone more than one investigation. 21 patients had X-ray taken, 43 patients Contrast Enhanced CT, 21 patients had an USS of the groin, 2 patients had MRI, and 1 patient had a Barium scan. In 18 (16%), decision to treat was based on clinical examination.
There is significant discussion around the establishment of preoperative diagnosis of De Garengeot’s hernia and more so regarding the diagnosis of appendicitis within it. Table 2 outlines the accuracy CE CT, when used, in establishing a pre-operative diagnosis in 74% cases. In the absence of discussion about CT imaging, it is assumed that CT was not used in the diagnostic work up.
Diagnostic ImagingNumber of CasesCE CT used for pre-operative diagnosisDiagnostic32Non-Diagnostic11Total43CE CT not used for pre-operative diagnosis69
Table 2: CE CT finding in reviewed data.
74 studies have reported appendicitis in the Femoral Hernia. Table 3 outlines the histological outcome of the appendix. Of the 87 cases that have reported the histopathology, 12 (14%) were normal appendixes within a De Garengeot’s.
Histological OutcomeNumber of Reported CasesNormal Appendix [22,24,35,41,44,49,54,55,75]12Acute Appendicitis 61Not Reported25Perforated Appendicitis 8Necrotic Appendicitis [51,85]2Appendiceal Abscess 1Chronically relapsing Appendicitis 1Ischemic Gangrenous Appendicitis 1Total No of Patients111
Table 3: Histological outcomes in reviewed studies.
The surgical technique is most often determined by the diagnosis of Femoral Hernia. Of interest in this study were the subsequent appendicectomy and the implications of encountering De Garengeot’s hernia, especially with an inflamed or infected appendicitis within. In the current collection of cases, most of the surgeries were determined by the surgeon’s preference to access the hernia, with the Lockwood approach being the most employed (n=35) in this review. In 64% of the cases (n=72), the appendix was removed through the initial incision. Table 4 outlines the initial surgical approach, the procedure for the subsequent appendicectomy performed and the surgical approach for hernia repair.
Hernia Repair/Appendicectomy Approach/Approach to HerniaNumber of Reported CasesHerniorrhaphy332Appendicectomy via initial incisionMcEvedy Approach3Laparotomy4Lotheissen's approach1King's College approach1Laparoscopic Approach1Approach Not Specified4Laparoscopic appendicectomyLotheissen's approach2Laparoscopic Approach1Approach Not Specified1Laparotomy for AppendicectomyLaparoscopic Approach1Approach Not Specified8No AppendicectomyLotheissen's approach1HemicolectomyApproach Not Specified1Appendicectomy Technique Not SpecifiedApproach Not Specified3Hernia Repair by McVay Technique422Appendicectomy via initial incisionLockwood approach6Lotheissen's approach5McEvedy Approach2Approach Not Specified1Appendicectomy via initial incisionMcEvedy Approach2Lotheissen's approach1Laparoscopic appendicectomyLaparoscopic Approach1Laparotomy for Appendicectomy3Lockwood approachLotheissen's approach1Herniorrhaphy through low approach520Appendicectomy via Initial incisionLockwood approach12Lotheissen's approach3Appendicectomy via Lanz incisionLockwood approach1Laparotomy for AppendicectomyLockwood approach3Incidental AppendicectomyLockwood approach1Mesh Hernia Repair620Appendicectomy via initial incisionLockwood approach8Lotheissen's approach6Gregoire incision1Approach Not Specified2Laparoscopic appendicectomyLaparoscopic Approach1Lotheissen's approach1Laparotomy for AppendicectomyApproach Not Specified1Herniorrhaphy through high approach74Appendicectomy via initial incisionLotheissen's approach1McEvedy Approach2Laparotomy for AppendicectomyMcEveedy Approach1TAP Femoral Hernia Repair82Laparoscopic appendicectomyLaparoscopic Approach1Laparoscopic TAP Approach1Herniorrhaphy through Lichenstein Technique91Appendicectomy via initial incisionApproach Not Specified1TEP Hernia Repair101Laparoscopic appendicectomyLaparoscopic TEP Approach1Lockwood Repair of Femoral Hernia111Appendicectomy via Lanz incisionLockwood approach1De Oliveira Hernia Repair121Appendicectomy via initial incisionLotheissen's approach1Hernia Repair Not Specified7Appendicectomy Technique Not SpecifiedApproach Not Specified7Grand Total10913
13 Two case reports did not provide details about surgery
Table 4: Surgical approach and procedures in reviewed studies.
In retrospect, there is value in noting the impact of the preoperative diagnosis on the approach decision. With a limited number of cases that have been diagnosed specifically as appendicitis within a De Garengeot’s, Table 5 compares the preoperative diagnosis to the impact of the surgical approach in whether the appendix could be accessed through the initial incision.
Pre-Operative Diagnosis and Surgical ApproachNumber of PatientsNo Preoperative DiagnosisAppendicectomy via Initial incision22Separate Incision for Appendicectomy6Laparoscopic appendicectomy3Not Specified1CE CT- De Garengeot's14Appendicectomy via initial incision19Separate Incision for Appendicectomy7Laparoscopic appendicectomy1Not Specified5USS- Femoral Hernia with Bowel Obstruction15Appendicectomy via initial incision6Separate Incision for Appendicectomy2Laparoscopic appendicectomy1CE CT- Strangulated/ Incarcerated Hernia16Appendicectomy via initial incision6Laparotomy for Appendicectomy1AXR- No Bowel Obstruction17Laparoscopic appendicectomy1Appendicectomy via initial incision3Laparotomy for Appendicectomy2AXR- Bowel Obstruction18Appendicectomy via initial incision5Laparoscopic appendicectomy1USS- De Garengeot's19Appendicectomy via initial incision4Separate Incision for Appendicectomy3Laparoscopic appendicectomy1CE CT- Femoral Hernia, No Obstruction20Appendicectomy via Initial incision4MRI- De Garengeot's21Appendicectomy via initial incision2USS- Femoral Hernia, No Bowel22Laparoscopic appendicectomy2Barium Scan- Appendico-Cutaneous Fistula23Appendicectomy via initial incision1Total Number of Patients111
15 [19, 28, 31, 43, 49,55,61]
16 [17, 46, 49, 50, 61,65,81]
17 [11, 25,36, 52,83]
19 [21,22, 41,55,67]
Table 5: Pre-operative diagnosis vs. surgical approach in reviewed studies.
There was one reported post-operative death, and of the case reports with post-operative morbidity, 11 patients reported postoperative complications. Of these, one patient had mobility issues due to Parkinson’s disease , unrelated to surgery and has been excluded. The post-operative death occurred in a patient (76/F) that presented with diabetic ketoacidosis. The mean postoperative stay was 5 (1-22) days.
A Case Sample
An 86-year-old man presented with 48-hour history of progressive nausea, vomiting and right lower abdominal and groin pain. He had also not opened his bowels without passage of flatus for 24-hours. Medical history included hypertension, Chronic Obstructive Pulmonary Disease, Congestive Cardiac Failure, multiple chest infections, and a left hip replacement. Although WHO performance status was 3, his walking was limited to 100 yards with exertional dyspnoea.
Physical examination showed a mildly distended abdomen and an 8 × 6 cm tender swelling at the right inguinal ligament with no associated skin changes. Laboratory investigations revealed WCC>12 and CRP>70. Plain Abdominal X-Ray exhibited a few loops of gas-fluid filled small bowel, consistent with small bowel obstruction. A clinical diagnosis of obstructed Femoral Hernia was made. This was a high-risk case that was undertaken under spinal anaesthesia following discussion with consultant anaesthetist. A high McEvedy approach was undertaken and following reduction of the Femoral Hernia, the sac revealed an inflamed long, thick, coiled appendix. Conventional appendicectomy was performed followed by the closure of the peritoneum. Femoral ring was approximated with 1 Ethibond sutures. Patient made a slow but uneventful recovery and was discharged home after 7 days. Histology confirmed acute appendicitis.
Current literature quotes incidence between 0.8% to 0.13% that was first reported by Ryan WH in 1937, following observation and data collation of 8,692 cases of appendicitis with the appendix identified only in 11 (0.13%) hernia sacs .
The exact pathogenesis of De Garengeot’s is still debatable. Femoral Hernias are four times more common in females , and more so in post-menopausal women. There is a school of thought that during embryological development differing degrees of rotation of the alimentary tract could result in the abnormal attachment of the vermiform appendix to the pelvis, resulting in a ‘pelvic appendix’ risking herniation through the femoral ring.
Appendicitis within a Femoral Hernia does not usually present as a classical acute appendicitis, thus, limiting the scope of diagnosis (appendicitis independently). The detection of the irreducible hernia is, in itself, an indicator of surgery and historically literature reports exploration of the hernial sac confirming its contents. Powell HD et al. , reports pre-operative diagnosis of appendicitis within a femoral sac by examination alone and Voitk AJ et al.  were one of the first authors preoperatively reporting appendico-cutaneous fistula in a Femoral Hernia  in a chronic case of a groin swelling of 15 years.
Preigo et al.  observed that presentations in these cases are atypical and even computersied tomography was not accurate in all cases. However, Abdul-Ghaffar S et al.  report 100% sensitivity and 98.9% specificity for computerised tomography as a pre-operative diagnostic modality for acute appendicitis in De Garengeot’s hernia. Salkade PR  and Ikram S et al.  also report similar results with computerised tomography. Filatov J et al.  and Park HR et al.  provide supportive arguments in using ultrasonography as a diagnostic modality of choice, given its accuracy, cost effectiveness, and accessibility compared to computerised tomography or MRI . Ultrasonography risks operator bias as the operator may not be trained to identify the hernial sac contents once the diagnosis of herniation is established. Halpenny D et al.  have reported successful use of MRI for establishing a pre-operative diagnosis.
Following review of the published literature in 2013, Kalles V et al.  noted computerised tomography to be diagnostic in 44% of the patients evaluated across 31 case reports. In this systematic review, computerised tomography showed 74% accuracy in diagnosing appendicitis within De Garengeot’s hernia.
From the data in the current study, the use of CT seems to provide pre-operative diagnosis, allowing the surgeon to make a more informed decision around surgical approach.
There is no consensus regarding the surgical approach to De Garengeot’s hernia. A major aspect of the decision making was affected by the pre-operative diagnosis and the surgeon’s priority to explore the abdomen to prevent worsening of any incarceration. In cases where a pre-operative diagnosis of De Garengeot’s was established (with or without appendicitis), the approach was based on the need that both the appendix and the femoral ring are accessible through single incision. The use of laparoscopy in the event of no pre-operative diagnosis is supported by Comman A et al.  to reduce incidence of postoperative ileus. The suitability of TAPP over TEP due to the potential benefit of evaluating intraabdominal and hernial contents is also noted, with Al-Subaie S et al.  favouring the use of TAPP over TEP as a diagnostic tool for a patient clinically diagnosed with a irreducible right Femoral Hernia. This is countered by Beysens M et al.  and Shiihara M et al. , who argue that due to the containment of the site of appendicectomy, a mesh repair can be done without risk of infection. There is discussion around the use of interval hernia repair, where the appendicectomy was initially done followed by the hernioplasty. Use of mesh is extensively debated across the literature, primarily for the risk of infection especially in a De Garengeot’s hernia with inflamed or infected appendix.
In the current review, of the 109 cases that specified the surgical techniques, the most common technique for hernia repair was suturing with non-absorbable material (n=86), and another 20 had mesh repair. The only reported post-operative complication in patients with mesh hernia repair identified was wound infection (n=3). When the decision for hernia repair was done without a mesh, the most common technique used were interrupted sutures (n=58) and McVay’s repair (n=26).
Three case reports describe unusual surgical techniques. Mizumoto R et al.  chose the King’s College approach where a single skin incision allowed exploration of the groin hernia and its contents and the subsequent entry into peritoneum for appendicectomy. De Oliveira technique for hernia repair as described by Couto HS et al. , explain that the choice was made due to its effectiveness in hernia repair without the need of mesh and its cost effectiveness. Lacaille-Ranger A et al.  used modified Nyhus technique, where they describe posterior approach to hernia repair by dividing the layers of the abdominal wall to expose the femoral ring from the preperitoneal space.
The post-operative outcomes show a 9.8% of the cumulative patients reporting post-operative complications, with seven patients reporting wound infection. The current study is limited in the ability to evaluate the relationship between the pre-operative surgical decision and the post-operative outcome. There is a lower incidence of post-operative morbidity (1%) in patients that have had an accurate pre-operative diagnosis; however, this is not conclusive given the limited number of patients (32) and the lack of a control group to compare [80-87].
The study is limited in that there is not enough data to establish a causal relationship between the pre-operative surgical decision and the post-operative outcomes. Without a structured casecontrol study, any correlational conclusions remain speculative.
Appendicitis within a De Garengeot’s hernia often lacks a classic presentation. The irreducible hernia becomes the point of focus and an indicator for surgery and the contents of the sac are only detected after direct exploration. There have been cases where a pre-operative diagnosis was established with the use of imaging, however there can be limitation based on their availability, cost, and operator training. A clinical scenario of irreducible, strangulated hernia warrants an urgent surgical exploration. However, if a pre-operative diagnosis can be established: laparoscopic exploration, sequestered appendicectomy and hernia repair could potentially be the appropriate option for the management of De Garengeot’s hernia. Current literature review confirms the rarity of De Garengeot’s hernia leading to the lack of a standardised operative technique and the authors feel the discretion for approach would depend on the surgeon’s preference and available expertise.31395